Patients’ experiences and perspectives of patient-reported outcome measures in clinical care: A systematic review and qualitative meta-synthesis

Background Patient-reported outcome measures (PROMs) or patient-reported outcomes (PROs) are used by clinicians in everyday clinical practice to assess patients’ perceptions of their own health and the healthcare they receive. By providing insight into how illness and interventions impact on patients’ lives, they can help to bridge the gap between clinicians’ expectations and what matters most to the patient. Given increasing focus on patient-centred care, the objective of this meta-synthesis was to summarise the qualitative evidence regarding patients’ perspectives and experiences of the use of PROMs in clinical care. Methods A systematic search of the following databases was undertaken in August 2020: Medline, EMBASE, EMCARE, PsychINFO, Scopus and the Cochrane Library. This review was conducted and reported in line with the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) statement. Methodological quality of the included studies was assessed using the Critical Appraisal Skills Programme checklist for qualitative research (CASP). A meta-ethnographic approach was used for data extraction and meta-synthesis of findings (PROSPERO registration: CRD42020202506). Results Fourteen studies from a range of countries with differing qualitative research methodologies were identified. Three themes were identified, namely ‘patient preferences regarding PROMs’, ‘patient perceived benefits’ and ‘barriers to patient engagement with PROMs’. The perspectives of patients suggested they preferred PROMs that were simple and relevant to their conditions and found benefits in the way they facilitated self-reflection and effective communication with their clinicians. Patients, however, questioned the relevance of some individual questions and purpose. Conclusion PROMs can be a useful tool in the clinical setting by enabling individualisation and patient centred care. This meta-synthesis provides insights into what patients find beneficial as well as barriers to their engagement, highlighting the importance of educating patients about PROMs.


Introduction
In response to growing recognition of the importance of patient-centred care, the last 30 years have seen a proliferation in the number of tools designed to assess patients' perceptions of their own health and the healthcare they receive. A patient-reported outcome (PRO) is "any report of the status of a patient's health condition that comes directly from the patient, without interpretation of the patient's response by a clinician or anyone else [1]." Patient-reported outcome measures (PROMs)-the instruments used to assess PROs-may capture the impacts of a health condition or its treatment on quality of life, functional status, and symptoms, as well as At the same time, numerous barriers to the effective use of PROMs in practice have been identified [15]. Regarding the characteristics of PROMs, barriers commonly reported by clinicians include concerns about their validity and reliability, perceptions about the value of PROMs (e.g., provision of clinically valuable information) and the design process (e.g., decisions about the choice of a PROM, and the processes for PROMs data gathering, management, utilisation and interpretation), and the perceived difficulty and cost of implementing them [8,11,[15][16][17]. Clinician-reported barriers pertaining to patient needs include the perceived relevance of PROMs, and concerns that they may add undue burden, cause distress, or detrimentally impact on patient care [8,11,[15][16][17]. There are also technical and administrative barriers to the successful implementation and translation of PRO monitoring into practice; for example, where there is no systematic approach for using PRO data to inform patients' treatment plans, or where PRO data cannot be easily integrated into electronic patient records [18]. It has been argued that this is because more attention has been given to the psychometric properties of PROMs than to how PRO data will improve the quality of patient care [6].
Qualitative research is vital for understanding the mechanisms and contextual factors that determine whether interventions are effective in practice and developing theoretical models to guide successful implementation [8]. Two reviews of qualitative research regarding health professionals' perceptions of PROMs have been published [8,19] but synthesis of the qualitative evidence regarding patients' perspectives and experiences of PROMs has not been conducted. To bridge this gap, this systematic review and meta-synthesis aimed to summarise the qualitative evidence regarding patients' perspectives and experiences of the use of PROMs in routine care.

Search protocol and registration
The protocol for this systematic review was registered with the international prospective register of systematic reviews-PROSPERO (Registration # CRD42020202506).

Inclusion criteria
This review included qualitative research that explored experiences and perspectives of patients regarding PROMs use in healthcare, and which used thick descriptions to describe these findings. The use of thick descriptions meant authors of the qualitative research were able to provide detailed and direct quotes about the findings rather than mere descriptive summaries, or mere quantification of qualitative data. Research, which was focused on clinicians' experiences of PROMs solely, and was not exclusively underpinned by qualitative research methodologies/methods were also excluded. All adult (i.e., over the age of 18) patient populations (i.e., any health condition or setting) were of interest. The review was limited to studies published in English.

Search strategy and study selection
An initial search was conducted in MEDLINE (PubMed), and then translated for and undertaken across all other included databases: Embase, Emcare, PsychINFO, Scopus, and the Cochrane Library (LC & PL). The databases were searched between 10 th and 28 th August 2020 with English language limit, but no restrictions placed on date of publication. To minimise publication bias, targeted keyword searches of grey literature sources (such as Google and organisational websites) were conducted, in conjunction with hand searching of the reference lists of included studies (pearling) (LC & PL). The search combined MeSH terms and key descriptors of 1) patients, 2) experiences and perspectives, 3) PROMs, and 4) qualitative research. The search terms and parameters are provided in Table 1. The search results were exported into Endnote™ where the hits from databases and grey literature were merged. These results were then transferred into Covidence™ (Covidence.org), which is a data management software used for systematic reviews. Following removal of duplicates, two independent reviewers (LC & PL) screened first by title and abstract, followed by full text screening. Any disagreements were resolved through discussion between the reviewers.

Critical appraisal
The methodological quality of the included studies was assessed using the Critical Appraisal Skills Programme checklist for qualitative research (CASP) [20], which is freely available and widely used to appraise qualitative research. Appraisal of validity was based on declaration of study aims and whether the chosen qualitative methodology was appropriate to address those aims [20]. Appraisal of bias was based on whether the relationship between the researcher and participants was considered [20]. Appraisal of ethical considerations was based on whether ethics committee approval was granted, and informed consent obtained [20]. Appraisal of data collection was based on whether data collection methods were appropriate and justified, and if the form of data was clear [20]. Appraisal of data analysis was based on clarity, rigour, and justification of the analytical approach (e.g., thematic analysis), whether biases were examined, and whether sufficient data were presented to support findings [20]. Appraisal of transferability was based on the applicability of the research to other contexts and whether adequate detail was given about the sample [20]. Studies were not excluded based on their methodological quality. However, this information was used to report, analyse and discuss the overall review findings. To ensure consistency of the critical appraisal process, independent appraisals of the studies were completed by all members of the review team (LC, CF, PH, PL & MS) and compared. Any disagreements were resolved through discussion between the review team.

Data extraction and synthesis
A meta-ethnographic approach [21] was used for data extraction and meta-synthesis of findings from the studies deemed eligible for inclusion. This approach has previously been used in health research, especially when exploring patient experiences of their own health and healthcare they received. Therefore, given that this systematic review aimed to summarise patients' perspectives and experiences of the use of PROMs in routine care, this approcah was an ideal choice. Meta-ethnography is therefore well-established [22], rigorous, and underpinned by three stages of analysis [23] For the first stage of analysis, one reviewer (MS) read the studies multiple times and extracted study characteristics, key findings, and quotes from participants from the included studies into a predefined template on Microsoft Word. This was particularly useful in becoming familiar with, and understanding, the content of the included studies and starting to recognise emerging themes. During the second stage of analysis, known as reciprocal translation, participant quotes from the included studies were grouped into concepts [23]. This was informed by a pragmatic (initial grouping of concepts were refined over time with consultations between reviewers) as well as a methodological (trying to maintain and staying close to the original data) approach. The third and final stage involved regular and systematic comparison and interpretation of the data, and synthesis into broad themes. As there is no one standard way to do this, this stage was informed by a previous systematic review which also used a meta-ethnographic approach [22].
To enhance rigour and minimise biases, the entire process was overseen and reviewed by another member of the review team (SK).

Search outcomes
The literature search yielded a total of 9261 articles, including 9258 records from database searches and a further three studies were identified through pearling. After the removal of duplicates, the titles and abstracts of 5706 articles were screened against the inclusion criteria in Covidence™. The title and abstract screen identified 39 potentially relevant studies for full text review. Of these, 25 studies were excluded for reasons documented in   [37], constant comparison [35] and Delphi consensus methods [29]. All of the studies involved face-to-face and/or phone semi-structured interviews, with the exception of four studies [28, 29, 33, 37] that collected patient experiences and perspectives via face-to-face focus groups.

Characteristics of included studies
The majority of the studies involved purposive sampling of adult male and female outpatients living with a chronic condition (including chronic kidney disease, inflammatory arthritis, depression, chronic haematological disease, Parkinson's disease, epilepsy and chronic musculoskeletal presentations), with sample sizes ranging from 9 to 32 individuals. One study included patients with acute musculoskeletal presentations [27]. Two studies did not report participant gender [25,26], and one also did not report participant age [26]. Two studies [24, 25] did not explicitly state that their sample comprised outpatients.
All of the studies collected participant experiences and perspectives of PROMs (digital and/ or hardcopy, condition-specific and/or generic) use. The PROMs either had been used in the past and/or were currently in use, or had been implemented as part of the study, with the exception of two studies [24, 28] that involved conceptual PROMs (i.e., the idea of implementing PROMs). The characteristics of individual studies are presented in Table 2.

Methodological quality
All of the studies were evaluated as having a clear statement of the aims of the research, an appropriate qualitative methodology, data collection that addressed the research question(s), sufficiently rigorous data analysis, and a clear statement of findings. Two studies [24, 25] did not explicitly state their research design. Three studies [27, 31, 36] had recruitment strategies that involved treating therapists identifying and directly inviting patients to participate, which was considered to be inappropriate. None of the articles included researcher declarations, but three [27, 36,37] included statements referring to management of the relationship between researchers and participants. The critical appraisal did not result in the exclusion of any studies or influence the meta-synthesis. The results of the critical appraisal are summarised in Table 3.   PROMs increased patients' sense of their "being taken more seriously" [32, 35,37].
"At least the questionnaire is more profound than the usual how-are-you-questions. It seems as if they take you a bit more seriously now than they did before." PROMs facilitated clinician planning, assessment, diagnosis, treatment and monitoring [26,27,30,35,37], which improved an individualised approach to patients [27, 35,37] and increased efficiency for patient visits [29]. This facilitation of processes was also reported as a patient benefit in the conceptual study by Aiyegbusi et al., [24].
"Obviously the benefit of using PROMs is that PTs can prepare themselves for my visit. Using the PROM results, your PT (physical therapist) can analyze what might trigger the health problem and think about the intervention they might use. At a later phase, when the PROMs are completed again, they could analyze the progression did the pain decrease or is it completely resolved?" [27] "[. . .] You can of course certainly end up with more accurate treatment by filling in this kind of questionnaire." [37] "These questions take out the fluff and other things that the doctors talk about and focuses them on the issue. I think it helps the doctor" [29] Patients reported PROMs improved patient-clinician communication by providing an "icebreaker" [29], structure and condition-specific vocabulary, highlighting priorities plus opening-up the discussion to broader issues [29, 32], which increases mutual understanding [37] and enables shared decision-making [26]. This improved communication was also reported as a patient-clinician relationship benefit in the conceptual studies by Aiyegbusi et al., [24] and Navarro-Millan et al., [28].
"Well, I think it is very nice. The questions are much more everyday questions. That makes it much easier to explain and describe how your epilepsy actually is. Because you try and you try to explain how it is and how it feels to your close ones and to the doctors, but it is so hard to explain in a way that normal people can imagine how your body experiences it." [32] "It was extraordinarily helpful when you sit down with your doctor and he says, "This is what I see. . ." and I say, "Well this is what I see. . .". And we talk about how we can improve those areas that I would like to see improved, or maybe manage better those areas that may not be able to be improved" Patients living with depression and/or anxiety appreciated that PROMs enabled them to communicate difficult information impersonally [30,31,35]. However, Dowrick et al., [35] and Primdahl et al., [33] reported some patients manipulated PROM data to minimise stigma and/or influence treatment. This PROM susceptibility to manipulation was also reported in the conceptual study by Aiyegbusi et al., [24]. "[. . .] Sometimes I don't want to tell people how I'm feeling. You know, pride or don't want to bring somebody else down. So this way it's a more impersonal way of answering the questions because you're just marking on paper; you're not actually telling somebody face-to-face." "You're more likely to lie, well I found I'm more likely to lie. . . Because I still find a lot of stigma attached to depression" [35] Theme 3-Barriers to patient engagement with PROMs. The barriers to patient engagement with PROMs identified cover a range of topics including patients' capacity to answer, question relevance and validity, patients' understanding of purpose and inconsistent clinical utilisation.
Some patients questioned whether or not they were qualified to answer PROMs [32-34].
"Well, we are not supposed to be doctors, we are not supposed to assess our own health, because there are some people who are professionally educated to do that" [32] Alternately, some patients questioned the relevance of individual PROM questions [32, 34] and/or their accuracy for truly capturing patient's symptoms and experiences [30, 33, 35]. Damman et al., [36] reported some patients thought PROMs didn't tell them anything they didn't already know or told them things they didn't want to know.

Discussion
This systematic review examined the findings of qualitative studies with thick descriptions exploring patients' experiences and perspectives of PROMs in healthcare. The meta-synthesis identified three overarching themes: patient preferences for PROMs, perceived benefits of PROMs, and perceived barriers to the use of PROMs. Regarding preferences for PROMs, relevance of content and specificity to the disease or condition were considered important, as was presentation in a simple and consistent format. However, there was variability in preferences for how, where, and how often PROMs should be administered. Patients' perceived benefits of PROMs related to gaining a sense of empowerment through self-reflection and providing information to assist with clinical planning, assessment, diagnosis, individualised treatment, decision-making, and monitoring. Barriers to engagement with PROMs reported by patients included patients' capacity to provide credible information, perceived relevance and utility of the questions, understanding of purpose, and concerns about how information gathered is applied clinically.
The value patients placed on the relevance and specificity of PROMs in the included studies is consistent with findings from other evidence syntheses. A scoping review [11] found that a key factor for successful implementation of PROMs was ensuring that measures addressed issues relevant to patients. Relevance and specificity were also considered key indicators of the value of PROMs by clinicians in a systematic review of qualitative evidence [19]. To ensure relevance, careful consideration should be given to instrument selection, prioritising those developed in consultation with the patient groups they represent to capture information that is most meaningful to them. The use of individualised PROMs, which allow patients to select or weight issues of most personal relevance, may be particularly useful when significant differences between patients in priorities and areas of concern are anticipated [38]. Improved precision and efficiency of PRO assessments may also be achieved by using PROMs that utilise computer adaptive testing (CAT) algorithms to select the most relevant items for the respondent. In addition to relevance, other factors that have been found to influence the implementation of PROMs include their length and complexity, the availability of translated and culturally meaningful versions, and respondents' comfort level with technology if completing them electronically [11].
The variability evident in patients' preferences for paper-based and online PROMs completion is consistent with findings from the realist synthesis of PROMs by Greenhalgh et al., [6], which found that patients expressed preferences for different types of PROMs and different modes of administration. As preferences may influence how individuals respond to and engage with PROMs, identifying them and adapting processes accordingly may optimise the potential benefits of completing PROM assessments. A systematic review of reviews examining factors that influence the implementation of PROMs found that a key enabler to designing the PROMs process in an organisation was ensuring adaptability to both the organisational context and to specific patients [15]. This requires awareness of patients' unique characteristics, needs, and resources [15], to ensure that PROMs are appropriate for, and equivalently applicable to, each individual [38]. Examples of individual factors that may influence patients' preferences for, interpretation of, and responses to PROMs include age, disability, cultural background, education, health literacy, and previous healthcare experiences [38].
Patients' capacity to complete PROMs has been previously identified as a barrier to the use of PROMs in cancer care [15]. However, while this meta-synthesis identified concerns about whether patients are qualified to accurately assess their own symptoms, other reasons identified in the literature include disability or difficulty reading and/or responding to questions, being too unwell, and difficulty recalling symptoms or remembering to complete PROMs [46]. These findings highlight the importance of ensuring PROMs are user-friendly and provided in conjunction with adequate support. Just as patients in the included studies expressed concerns about how PRO data is applied clinically, negative perceptions about the value and clinical usefulness of PROMs have been identified as barriers to their use by clinicians in qualitative research [19,46]. Therefore, efforts should be made to ensure clarity regarding the purpose of PRO data collection and how data will be used, that appropriate instruments are selected, and that patients and clinicians are informed of these details [46].
A strength of this review was the inclusion of mixed-methods studies with qualitative content in addition to qualitative studies, which enabled examination of findings from study designs often employed in research on the development and piloting of PROMs that may be excluded from other qualitative syntheses. The included studies were based in a range of countries and healthcare contexts and presented the views of various patient groups involved in the use of PROMs. In many cases, studies included well-established patient measures, but also included views about PROMs as a general concept.
While the review methods followed best practice guidance in the conduct of meta-analyses (PRISMA), there are several potentially limiting factors. Language and publication bias are likely present given that the review included only studies published in English. However, there were seven studies included from non-English speaking countries, suggesting the impacts of language bias were minimised. To reduce publication bias, searches of grey literature and reference list pearling were conducted but these did not identify any sources for inclusion. Although the search strategy was comprehensive, it was not an exhaustive search of all databases and all potential sources of grey literature, thus it is possible that relevant studies may have been missed (e.g. particular focus on patient perspectives of PROMs and excluding clinician perspectives). Limitations were also present in the included evidence base. As identified through critical appraisal, some studies did not provide a clear description of the research methodology, and in some the role of the researcher was potentially unclear, resulting in trustworthiness issues.
This meta-synthesis summarises qualitative evidence about the experiences and perspectives of patients regarding the use of PROMs in healthcare. The variability in preferences expressed by patients suggests that a one-size-fits-all approach to implementing PROMs may not be adequate, which is further supported by the value patients placed on the relevance and specificity of PROM content. To optimise the relevance of PROMs, careful consideration of the patient, context, and instrument may be required. The findings also highlight the importance of having a clear purpose for collecting PRO data and understanding of how data will be applied clinically and communicating this information to patients. The benefits of PROMs perceived by patients in the included studies aligned with literature indicating that PROMs can facilitate communication, individualisation of care, identification of concerns, and shared decision making. The evidence base identified in this review highlights the need for more indepth inquiry into the perceived value and benefits of PROMs (e.g which patient groups are most likely to benefit from PROMs, optimal means of communicating the value of PROMs to patients etc), preferences for their delivery, and barriers to their use from the perspective of patients.
Supporting information S1 Table.